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DOI 10.1002/ajmg.a.61462

Медицинская библиотека · American journal of medical genetics. Part A · 2019

Growth charts in Kabuki syndrome 1.

Ruault Valentin, Corsini Carole, Duflos Claire, Akouete Sandrine, Georgescu Véra, Abaji Mario, Alembick Yves, Alix Eudeline, Amiel Jeanne, Amouroux Cyril, Barat-Houari Mouna, Baumann Clarisse, Bonnard Adeline, Boursier Guilaine, Boute Odile, Burglen Lydie, Busa Tiffany, Cordier Marie-Pierre, Cormier-Daire Valérie, Delrue Marie-Ange, Doray Bérénice, Faivre Laurence, Fradin Mélanie, Gilbert-Dussardier Brigitte, Giuliano Fabienne, Goldenberg Alice, Gorokhova Svetlana, Héron Delphine, Isidor Bertrand, Jacquemont Marie-Line, Jacquette Aurélia, Jeandel Claire, Lacombe Didier, Le Merrer Martine, Sang Kim Hanh Le Quan, Lyonnet Stanislas, Manouvrier Sylvie, Michot Caroline, Moncla Anne, Moutton Sébastien, Odent Sylvie, Pelet Anna, Philip Nicole, Pinson Lucile, Reversat Julie, Roume Joëlle, Sanchez Elodie, Sanlaville Damien, Sarda Pierre, Schaefer Elise, Till Marianne, Touitou Isabelle, Toutain Annick, Willems Marjolaine, Gatinois Vincent, Geneviève David

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Аннотация

Kabuki syndrome (KS, KS1: OMIM 147920 and KS2: OMIM 300867) is caused by pathogenic variations in KMT2D or KDM6A. KS is characterized by multiple congenital anomalies and neurodevelopmental disorders. Growth restriction is frequently reported. Here we aimed to create specific growth charts for individuals with KS1, identify parameters used for size prognosis and investigate the impact of growth hormone therapy on adult height. Growth parameters and parental size were obtained for 95 KS1 individuals (41 females). Growth charts for height, weight, body mass index (BMI) and occipitofrontal circumference were generated in standard deviation values for the first time in KS1. Statural growth of KS1 individuals was compared to parental target size. According to the charts, height, weight, BMI, and occipitofrontal circumference were lower for KS1 individuals than the normative French population. For males and females, the mean growth of KS1 individuals was -2 and -1.8 SD of their parental target size, respectively. Growth hormone therapy did not increase size beyond the predicted size. This study, from the largest cohort available, proposes growth charts for widespread use in the management of KS1, especially for size prognosis and screening of other diseases responsible for growth impairment beyond a calculated specific target size.

Для цитирования: Ruault Valentin, Corsini Carole, Duflos Claire, Akouete Sandrine, Georgescu Véra, Abaji Mario, Alembick Yves, Alix Eudeline, Amiel Jeanne, Amouroux Cyril, Barat-Houari Mouna, Baumann Clarisse, Bonnard Adeline, Boursier Guilaine, Boute Odile, Burglen Lydie, Busa Tiffany, Cordier Marie-Pierre, Cormier-Daire Valérie, Delrue Marie-Ange, Doray Bérénice, Faivre Laurence, Fradin Mélanie, Gilbert-Dussardier Brigitte, Giuliano Fabienne, Goldenberg Alice, Gorokhova Svetlana, Héron Delphine, Isidor Bertrand, Jacquemont Marie-Line, Jacquette Aurélia, Jeandel Claire, Lacombe Didier, Le Merrer Martine, Sang Kim Hanh Le Quan, Lyonnet Stanislas, Manouvrier Sylvie, Michot Caroline, Moncla Anne, Moutton Sébastien, Odent Sylvie, Pelet Anna, Philip Nicole, Pinson Lucile, Reversat Julie, Roume Joëlle, Sanchez Elodie, Sanlaville Damien, Sarda Pierre, Schaefer Elise, Till Marianne, Touitou Isabelle, Toutain Annick, Willems Marjolaine, Gatinois Vincent, Geneviève David. Growth charts in Kabuki syndrome 1.. American journal of medical genetics. Part A. 2019. DOI: 10.1002/ajmg.a.61462.

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